Indian Journal of Pathology and Microbiology

CASE REPORT
Year
: 2020  |  Volume : 63  |  Issue : 2  |  Page : 309--311

Cytological diagnosis of primary cutaneous histoplasmosis with hemophagocytosis in immunocompetent patient – A rare case from non endemic region


Smita Chandra1, Manveer K Raina1, Dushyant S Gaur1, Vinish K Agarwal2,  
1 Department of Pathology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Doiwala, Dehradun, Uttarakhand, India
2 Department of Otolaryngology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Doiwala, Dehradun, Uttarakhand, India

Correspondence Address:
Smita Chandra
Department of Pathology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Doiwala, Dehradun - 248 140, Uttarakhand
India

Abstract

Histoplasma capsulatum is an opportunistic dimorphic fungus caused by inhalation of spores present in the soil. It is extremely rare in the northern Himalayan region of India and is usually asymptomatic. It may rarely progress to disseminated histoplasmosis which is usually observed in immunocompromised patients associated with malignancy, acquired immunodeficiency syndrome or diabetes. The present case is being reported because of unusual cutaneous presentation of disseminated histoplasmosis in an immunocompetent patient of non-endemic region. The case also highlights the importance of fine needle aspiration cytology for its precise early diagnosis and avoiding of further complications.



How to cite this article:
Chandra S, Raina MK, Gaur DS, Agarwal VK. Cytological diagnosis of primary cutaneous histoplasmosis with hemophagocytosis in immunocompetent patient – A rare case from non endemic region.Indian J Pathol Microbiol 2020;63:309-311


How to cite this URL:
Chandra S, Raina MK, Gaur DS, Agarwal VK. Cytological diagnosis of primary cutaneous histoplasmosis with hemophagocytosis in immunocompetent patient – A rare case from non endemic region. Indian J Pathol Microbiol [serial online] 2020 [cited 2020 Jul 13 ];63:309-311
Available from: http://www.ijpmonline.org/text.asp?2020/63/2/309/282717


Full Text



 Introduction



Histoplasma capsulatum is an opportunistic dimorphic fungus caused by inhalation of spores present in the soil, bird and bat droppings.[1] It is commonly observed in United States of America, Africa while endemic cases from India have been reported from Eastern India and West Bengal.[2],[3] It is extremely rare in north Himalayan region of India and only limited cases have been reported from here in the literature.[4] It is usually asymptomatic and may rarely progress to disseminated histoplasmosis in only 5-10% of the cases.[1] Disseminated histoplasmosis is usually observed in immunocompromised patients associated with malignancy, acquired immunodeficiency syndrome (AIDS) or steroid therapy.[5] Fine needle aspiration cytology (FNAC) is an important diagnostic tool and close examination of smears for subtle cytomorphological features may be helpful for its early diagnosis.

The present case is being reported because of unusual cutaneous presentation of disseminated histoplasmosis in an immunocompetent patient of non-endemic region. The case also highlights the importance of fine needle aspiration cytology with vigilant examination of subtle cytomorphological features for its precise diagnosis.

 Case Report



A 37-year-old male patient, farmer by profession, presented with right lower thigh and right supraclavicular swellings – which were present for 20 days and associated with intermittent fever without cough. The swellings were non tender, in form of diffuse erythematous papules, firm and were of large size of about 5 × 3 cm [Figure 1]a and [Figure 1]b. There was no associated organomegaly and no history of diabetes mellitus, tuberculosis, or any relevant drug history. His blood counts and blood sugar levels were within normal limits. The serological test for Hepatitis B surface antigen and human immunodeficiency virus (HIV) were also negative. X-ray chest and ultrasound abdomen showed normal study. FNAC of the swellings were advised and cytological smears showed similar morphology for both swellings. The smears were cellular and showed mixed inflammatory infiltrate comprising of eosinophils, plasma cells, abundant histiocytes and macrophages in necrotic background [Figure 2]a. The smears also revealed intra and few extracellular fungal spores which were round to oval eccentric ring like surrounded by clear halo and cytological diagnosis of histoplasmosis was rendered [Figure 2]b and [Figure 2]c. Another important associated cytological feature which was observed was evidence of hemophagocytosis [Figure 2]d. The patient was immediately started on amphotericin B. Soon enough, he recovered and is now on follow up.{Figure 1}{Figure 2}

 Discussion



Histoplasma capsulatum infection usually does not present with any symptoms and only few infected persons (<1%) present with pulmonary or disseminated histoplasmosis.[1] Cutaneous lesions may be associated with disseminated histoplasmosis in less than 5% of cases and present as papule, cellulitis like lesions or ulcerated nodules.[6] Rarely cutaneous histoplasmosis may be the only manifestation of the disease without any other organ involvement and such cases of primary cutaneous histoplasmosis present as solitary self limiting ulcerated nodule.[6] However, this case presented with primary cutaneous histoplasmosis with 2 plaque-like lesions on the right neck and thigh region. Immunosuppression has been suggested to be associated with such type of presentation but our case of multiple cutaneous histoplasmosis presented in immunocompetent patient.

In India, histoplasmosis has been reported mostly from West Bengal and Orissa. It is extremely rare to observe the same in north Himalayan Uttarakhand region.[4] The present case was a farmer by occupation, which may be the factor responsible for infection as inhalation of spores from soil has been reported to be source of the infection.

It is essential to diagnose this multi lesion form of infection early, so as to prevent further dissemination and other complications. It is also essential to exclude other common differential diagnosis such as tuberculosis or malignancy for appropriate treatment. FNAC has proved to be an important diagnostic tool for early and easy precise diagnosis. The smears reveal intra and extra cellular 2-4μ oval eccentric ring like organisms which have clear halo around them [Figure 2]. Other organisms which may mimic its cytological picture include leishmaniasis or candidiasis. However, Leishmania will show a kinetoplast present in the small intracellular parasite while candidiasis will also show the presence of pseudohyphae which are absent in histoplasmosis. Cryptococcus may also mimic histoplasma, but it will be seen as narrow-based budding yeast surrounded by capsular halo. In addition to this, there are certain associated features which may also be helpful in clinching the diagnosis. Eosinophilia, plasmacytosis may suggest the infection and appreciation of hemophagocytosis (engulfment of neutrophils, red blood cells, plasma cells or lymphocytes) by macrophages may give an important clue for its presence.[7] Our case also demonstrated this important feature which was helpful in the diagnosis. Culture and direct KOH mount may also be helpful in diagnosis. Malhotra et al. have reported a case of cutaneous histoplasmosis to demonstrate the importance of direct KOH mount with negative culture for diagnosing histoplasmosis.[8] Amphotericin B is recommended for its treatment and our case also responded to it.[9]

Thus to conclude, primary cutaneous histoplasmosis is an unusual presentation of this infection which may be present at multiple sites even in immunocompetent patient. Fine needle aspiration cytology proves to be an important diagnostic tool for its identification. It is essential to vigilantly examine the smears for subtle cytomorphological features, such as hemophagocytosis for precise diagnosis even in non-endemic areas. An early definite diagnosis with prompt treatment would be helpful to avoid further complication.

Authors' contributions

SC: Conception and design of study, data acquisition, data analysis and manuscript editing.

MKR: Literature search, data analysis, manuscript preparation.

DSG: Intellectual input and analysis of data, manuscript editing and review.

VKA: Clinical and intellectual analysis of data, manuscript review.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1Kauffman CA. Histoplasmosis: A clinical and laboratory update. Clin Microbiol Rev 2007;20:115-32.
2Nair SP, Vijayadharan M, Vincent M. Primary cutaneous histoplasmosis. Indian J Dermatol Venereol Leprol 2000;66:151-3.
3Santosh T, Kothari K, Singhal SS, Shah VV, Patil R. Disseminated histoplasmosis in an immunocompetent patient-utility of skin scrape cytology in diagnosis: A case report. J Med Case Rep 2018;12:7.
4Chandra H, Chandra S, Sharma A. Histoplasmosis on bone marrow aspirate cytological examination associated with hemophagocytosis and pancytopenia in an AIDS patient. Korean J Hematol 2012;47:77-9.
5Gupta N, Arora SK, Rajwanshi A, Nijhawan R, Srinivasan R. Histoplasmosis: Cytodiagnosis and review of literature with special emphasis on differential diagnosis on cytomorphology. Cytopathology 2010;21:240-4.
6Weedon D. Mycoses and algal infection. In: Weedon's Skin Pathology. 3rd ed. India, London: Churchill Livingstone Elsevier; 2010. p. 595-6.
7Chandra H, Chandra S, Kaushik RM, Bhat NK, Shrivastava V. Hemophagocytosis on bone marrow aspirate cytology- Single centre experience in North Himalayan region of India. Ann Med Health Sci Res 2014;4:692-6.
8Malhotra S. Dhundial R, Kaur NJK, Kaushal M, Duggal N. Cutaneous histoplasmosis and role of direct microscopy- A case report. Clin Microbiol 2017;6:2.
9Rana C, Krishnani N, Kumari N. Bilateral adrenal histoplasmosis in immunocompetent patients. Diagn Cytopathol 2011;39:294-6.